Occipital neuralgia is a form of headache that involves the posterior occiput in the greater or lesser occipital nerve distribution. Pain can be severe and persistent with conservative treatment. We present a case of intractable occipital neuralgia that conventional therapeutic modalities failed to ameliorate. We speculate that, in this case, the cause of headache could be the greater occipital nerve entrapment by the obliquus capitis inferior muscle. After steroid and local anesthetic injection into obliquus capitis inferior muscles under fluoroscopic and sonographic guidance, the visual analogue scale was decreased from 9-10/10 to 1-2/10 for 2-3 weeks. The patient eventually got both greater occipital neurectomy and partial resection of obliquus capitis inferior muscles due to the short term effect of the injection. The successful steroid and local anesthetic injection for this occipital neuralgia shows that the refractory headache was caused by entrapment of greater occipital nerves by obliquus capitis inferior muscles.